J HEALTH POPUL NUTR

2014 Jun;32(2):372-376 ©INTERNATIONAL CENTRE FOR DIARRHOEAL

ISSN 1606-0997 | $ 5.00+0.20 DISEASE RESEARCH, BANGLADESH

CASE STUDY

Cutaneous Leishmaniasis in an Immigrant

Saudi Worker: A Case Report
Hafizur Rahman1, Mohammad A. Razzak1, Bikash C. Chanda1,
Khondaker R.H. Bhaskar1, Dinesh Mondal2
1
Clinical Laboratory Services, Diagnostic Labs, Laboratories, icddr,b; 2Centre for Nutrition and Food Security,
Parasitology Laboratory, Laboratories, icddr,b, GPO Box 128, Dhaka 1000, Bangladesh

ABSTRACT

Cutaneous leishmaniasis (CL), an uncommon disorder in South-East Asia, including Bangladesh, often
presents as granulomatous plaque on the exposed areas, with a high index of suspicion required for
diagnosis. Here we report the first imported case of CL caused by Leishmania tropica in a migrant Bang-
ladeshi worker in the Kingdom of Saudi Arabia (KSA). The case, initially suspected as a case of cutane-
ous tuberculosis, arrived at specimens reception unit (SRU) of diagnostic labs of icddr,b being referred
by the physician for ALS testing for tuberculosis. At his arrival in the SRU, one of the health personnel
of the unit who used to work in KSA suspected him as a case of CL. The diagnosis was confirmed by
smear microscopy which revealed plenty of amastigotes within macrophages. PCR was performed to
confirm the species. He was treated with sodium stibogluconate at Shahid Suhrawardy Medical College
Hospital, Dhaka.

Key words: Cutaneous leishmaniasis; LD bodies; Leishmania tropica; Sodium stibogluconate; Bangladesh

INTRODUCTION mens reception unit of diagnostic lab of icddr,b in

Tropical infections caused by Leishmania spp. can
present diagnostic problems both to physician as
well as the dermatologist. The clinical diagnosis is
not difficult with typical features of leishmaniasis
in endemic countries. However, in non-endemic
countries where cutaneous leishmaniasis (CL) is
not common as in Bangladesh, it can easily be
missed. When considering a cutaneous lesion of
possible infective cause, the common differentials
would include mycobacterial and deep fungal in-
fections. In a country like ours where tuberculosis
is more prevalent, cutaneous leishmaniasis is very
likely to be mistreated as cutaneous tuberculosis, es-
pecially lupus vulgaris. Here we report a case of cu-
taneous leishmaniasis who presented to the speci-
Correspondence and reprint requests:
Dr. Hafizur Rahman
Clinical Haematology & Microscopy Lab,
Clinical Laboratory Services
Diagnostic Labs, Laboratories
icddr,b
68, Shaheed Tajuddin Ahmed Sarani
Mohakhali, Dhaka 1212
Bangladesh
Email: hafizur@icddrb.org
December 2011 and was probably imported from
Kingdom of Saudi Arabia where the patient used to
work in the past.
CASE REPORT
A previously well, 37-year old young adult present-
ed to the SRU of diagnostic labs of icddr,b, with a
6-week history of skin lesions on his nose, ear, arm,
and fingers. He was referred to the SRU of diagnos-
tic labs for ALS testing for tuberculosis, along with
culture for pus from wound. However, the health
personnel in the SRU suspected the case as a pos-
sible case of CL based on his experience when he
used to work in KSA. Then, the patient was referred
to one of the experts of icddr,b in this field. Physi-
cal examination revealed painless erythematous
papules and nodules with overlying scale and
crust, some of which had central ulceration (Figure
1A and 1B) [The patient provided his written con-
sent to the authors to use his photograph in this
case study]. The patient was from Saudi Arabia, an
area in which cutaneous leishmaniasis is endemic.
He remembered being bitten by sandflies during
his stay at Saudi Arabia. Following the bite, the le-
Cutaneous leishmaniasis
Figure 1. (A) Lesion before treatmentand; (B) Lesion healed with scarring after 10 days of treatment
1A
sion started as an itchy red papule slowly enlarged
into an inflammatory papule to an ulcer. The le-
sions usually appear in non-covered regions of the
body, mainly the face, nose, ear lobules, elbows,
and fingers. The incubation period could not be
confirmed from history; it was between 3 and 4
weeks. Systemic examination was unremarkable.
The ulcer failed to heal, despite several course of
systemic antibiotics. There was no past medical or
drug history of note. Based on history and clini-
cal examination, a provisional diagnosis of CL was
made. Thin smear from dermal scrapings revealed
large macrophage containing abundant intracel-
lular Leishman-Donovan bodies (amastigotes); some
were free-lying in the dermis without any granu-
loma (Figure 2); tissue culture and polymerase
chain reaction (PCR) confirmed infecting agent as
Leishmania tropica (Figure 3). He was admitted to
Sir Salimullah Medical College Hospital (SSMCH)
and treated with sodium stibogluconate (SSG). In-
travenous SSG was given at a dose of 20 mg/kg/day
for 28 days, along with intralesional injection at a
dose of 30 mg/day/lesion for 10 days. A significant
improvement was observed after 10 days, and all
the ulcers were healed. After 10 days, the patient re-
ceived only intravenous SSG for another 18 days.
DISCUSSION
Leishmaniasis is a poverty-related disease caused by
several species of the genus Leishmania. It affects the
Volume 32 | Number 2 | June 2014
Rahman H et al.
1B
poorest of the poor and is associated with malnutri-
tion, displacement, poor housing, illiteracy, gender
discrimination, weakness of the immune system,
and lack of resources. Leishmaniasis is also linked
to environmental changes, such as deforestation,
building of dams, new irrigation schemes and ur-
banization, and the accompanying migration of
non-immune people to endemic areas. Each spe-
cies tends to occupy a particular zoo-geographical
zone (1). The clinical manifestations of leishmania-
sis depend on the interaction between the charac-
teristic virulence of the species and the host’s im-
mune response (2). These are transmitted by the
bites of female sandflies of the genus Phlebotomonas
in the Old World and Lutzomyia in the New World.
More than 20 species of Leishmania, pathogenic for
humans and other mammals, have been identified
worldwide (3). About 30 species of sandflies are
proven vectors; the usual reservoir hosts include
humans and domestic/wild animals. The definitive
diagnosis depends on demonstration of the para-
sites by smears, culture, PCR, and histological ex-
amination of suspected specimens.
The geographical distribution of leishmaniasis is ex-
tremely wide; it is prevalent on our four continents
and considered to be endemic in 88 countries, 67
of these being in the Old World and 21 in the New
World, including Bangladesh, Brazil, Afghanistan,
Iran, Saudi Arabia, Peru, Sudan, and India (4,5).
Leishmaniasis in the Old World is endemic in the
373
Cutaneous leishmaniasis Rahman H et al.

Figure 2. Dermal skin scrapings: (2A) Wright’s and (2B) Leishmann’s stains

2A 2B

2A. Dermal skin scrapings Wright’s stain. Plenty of LD 2B. Dermal skin scrapings Leishmann’s stain.
bodies (amastigotes) within a macrophage (x100) Amastigotes are seen within a macrophage (x100)

Figure 3. PCR of amplified ITS1 products for detection of L. donovani and L. tropica

Digestion of amplified ITS1

products with restriction
endonuclease HaeIII separated
in 1.5% agarose gel electropho-
resis for 3 hours

1=L. donavani
3, 4=L. tropica
2, 7=Negative control
5=reference strain—L. tropica
6=reference strain—L. donavani
8=123 bp molecular size marker

374 JHPN
Cutaneous leishmaniasis
Mediterranean Sea and the neighbouring coun-
tries. The annual incidence worldwide is about
400,000 cases, with a prevalence of approximately
350 million people infected (6). More than 90% of
cutaneous leishmaniasis worldwide can be found
in Afghanistan, Iran, Saudi Arabia, Syria, Brazil, and
Peru. Majority of the cases of cutaneous leishma-
niasis are found in adult men between 20 and 40
years (6). Tourists and workers from endemic areas
have an increased incidence of CL. CL is mostly
imported to endemic countries by immigrants and
returning travellers.
Human leishmaniasis is usually classified as viscer-
al, mucosal, or cutaneous. The different forms of
the disease are distinct in their causes, epidemio-
logical features, transmission, and geographical dis-
tribution. Visceral leishmaniasis (VL) or kala-azar is
caused by L. donovani, L. infantum, and L. chagasi.
These species, in contrast with the other species of
Leishmania that infect man, are normally viscero-
tropic and cause a severe systemic infection, often
accompanied with gross splenomegaly, anaemia,
diarrhoea, hepatomegaly, lymphadenopathy, and
signs of malnutrition (7). However, a certain per-
centage of VL may present as post kala-azar dermal
leishmianiasis (PKDL) generally after 2-3 years fol-
lowing the treatment for VL, which appears to com-
pletely remit. This PKDL also causes a diagnostic
dilemma in endemic countries. Mucosal leishma-
niasis (ML) is an uncommon but serious manifesta-
tion of Leishmania infection, resulting from haema-
togenous metastases to the nasal or oropharyngeal
mucosa from a cutaneous infection. It is usually
caused by parasites in the L. (Vianna) complex. Ap-
proximately half of the patients with mucosal le-
sions have had active cutaneous lesions within the
preceding 2 years but ML may not develop until
many years after resolution of the primary lesion.
ML occurs in <5% of individuals who have or had
localized cutaneous leishmaniasis caused by L. (V.)
braziliensis. Cutaneous leishmaniasis (CL) is mainly
caused by L. tropica, L. major, and L. aetiopica (8).
CL is also known as ‘Aleppo boil’, ‘Baghdad boil’,
‘Bay sore’, ‘Biskra button’, ‘Chiclero ulcer’, ‘Delhi
boil’, ‘Kandahar sore’, ‘Lahore sore’, ‘Leishmaniasis
tropica’, ‘Oriental sore’, ‘Pian bois’, and ‘Uta’ in re-
spective areas (9). The incubation period in CL is
usually measured in months but ranges from a few
days to over a year. In our patient, the lesion ap-
peared 3 to 4 weeks after bite of sandflies. He was
treated with intravenous and intralesional sodium
stibgluconate (850 mg daily) for 28 days to which
he responded well. The differential diagnosis is ex-
tensive and includes infective granulomas, such
Volume 32 | Number 2 | June 2014
Rahman H et al.
as lupus vulgairs, deep fungal infections, myco-
bacterium infections, leprosy, sarcoidosis, and sq-
uamous cell carcinoma. A high index of suspicion
is required for provisional diagnosis (10). Many
leishmania species and subspecies of the Leishma-
nia protozoa have different virulence and clinical
predilections; so, treatment should be tailored for
every individual. Old World disease tends to be self-
limiting. Leishmaiasis caused by this species does
not necessarily need to be treated unless the lesion
is in a cosmetically- or functionally-sensitive site.
In the New World, leishmaniasis treatment is very
often the standard of care because of high recur-
rence rate of chronic ulcers, recidivant lesions, or
mucocutaneous involvement.
Treatment of CL is often difficult. Multiple treat-
ment options are used throughout the world for
cutaneous disease. Besides oral and parenteral
medications (pentavalent antimonials, liposomal
amphotericin B, miltefosine, and some others), lo-
cal cryotherapy, intralesional infiltration of sodium
stibogluconate, local heat therapy, and various top-
ical paromomycin preparations are in practice for
many many years.
Antimonials are still the first-line drug in the treat-
ment of CL. Sodium stibogluconate (Pentostam)
and meglumine antimonite glucantime are es-
sentially similar drugs which contain pentavalent
antimony (Sb). Sodium stibogluconate can be ad-
ministered intravenously or intramuscularly while
meglumine antimonite should only be given via
the intramuscular route. The recommended dose is
20 mg/kg/day for 20-28 days (8). Treatment with
antimonials is associated with some side-effects,
such as myalgia as well as possible liver or cardio-
vascular toxicity, which fortunately is rare. A recent
study using intralesional sodium stibogluconate
showed that alternate daily or weekly administra-
tion of intralesional sodium stibogluconate was ef-
fective in the treatment of CL (10). Dapsone and
allopurinol have also been used for the treatment
of CL. The mechanism is unclear, although basic
biomedical studies have shown that Leishmania
cannot make all of their own nucleic acids and,
thus, it uses the host’s purine through the purine
salvage pathway (1).
Besides systemic treatment, local measures, such as
cryotherapy, local excision of a small focus and top-
ical treatment using 15% paromomycin ointment,
have also been shown to be effective in some cases
(1,11). Vaccines for prophylaxis and immunothera-
py have been developed and are currently undergo-
ing trials in many countries, including Venezuela,
375
Cutaneous leishmaniasis
Brazil, and Iran (1,10). The development of molec-
ular biology techniques is also improving knowl-
edge on the structure, evolution, and expression of
the Leishmania genome, and the study and defini-
tion of the mechanisms that regulate the parasite’s
biochemical and molecular features will certainly
contribute to the development of new and more
effective strategies for leishmaniasis treatment. So
far, chemotherapy with systemic and intralesional
sodium stibogluconate is effective without any ma-
jor side-effects as was seen in this case.
Visceral leishmaniasis and its skin complication
(PKDL) among those treated for VL are common
in Bangladesh. Fortunately, there is no report of CL
in Bangladesh. This could be due to the absence
of vector P. papattasi which might be the natural
selection and native CL patients (12). However,
this should be recalled that there was no reported
case of infected vector of dengue fever before 2000
when the first outbreak of dengue fever took place
in Bangladesh. Thus, the disease control authority
of Bangladesh should be alert about the imported
cases of CL. This case report indicates that, if health
workers in the immigration are experienced or
trained in finding of CL, they can easily be identi-
fied at arrival and referred for diagnosis and treat-
ment. This will reduce suffering of such patients;
and also lower the risk of disease transmission in
community through early case detection and prop-
er management.
Conclusions
Cutaneous leishmaniasis is a rare disease in Bang-
ladesh. Because of higher rate of travel and work
abroad, increased number of sporadic cases of cuta-
neous leishmaniasis in non-endemic areas should
be taken into account. The case emphasizes the
point that, when assessing lesions of possible infec-
tive aetiology, a detailed travel history and knowl-
edge of the common infective agents in the loca-
tion concerned are of great importance in arriving
at a correct diagnosis for appropriate treatment.
ACKNOWLEDGEMENTS
Authors gratefully acknowledge icddr,b and all of
its core donors which provide unrestricted support
to icddr,b for its operations and research. Current
donors providing unrestricted support include:
Australian Agency for International Development
376
Rahman H et al.
(AusAID); Government of the People’s Republic of
Bangladesh; Canadian International Development
Agency (CIDA); Swedish International Develop-
ment Cooperation Agency (Sida); and the Depart-
ment for International Development (DFID), UK.
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